Original article 207
Oblique pelvic osteotomy in the treatment of bladder
exstrophy in neonates
Marco Giordanoa, Antonio Di Lazzaroa, Vincenzo Guzzantia,d, Silvia Careria,
Pietro Bagolanb, Antonio M. Zaccarac and Renato M. Tonioloa
Bladder exstrophy is a congenital and rare malformation of
the lower abdominal wall with exposure of the bladder
mucosa to the external environment, and it is related to pelvis
abnormalities. Eighteen patients with bladder exstrophy were
treated with bilateral oblique pelvic osteotomy in conjunction
with urologic reconstruction after they were stabilized by cast.
No failure of midline closure was observed (wound
dehiscence or recurrence of bladder exstrophy). Follow-up
showed no leg length discrepancy or problems in walking.
Bilateral oblique pelvic osteotomy is a safe procedure to treat
bladder exstrophy, and it results in good orthopedic and
urological function. J Pediatr Orthop B 28:207–213 Copyright
© 2019 Wolters Kluwer Health, Inc. All rights reserved.
Introduction
Bladder exstrophy is a rare congenital malformation, with a
prevalence of one per 40 000 live births and a male : female
ratio of 2–3 : 1 [1–3]. It is the most frequent clinical expression
of an embryological malformation of the urogenital apparatus
and it ranges from the mildest form, epispadias (opening of
the urethral meatus on the dorsal side of the penis), to the
most severe form, cloacal exstrophy, in which the bladder
pathology is associated with intestinal prolapse and other
serious birth defects, with an incidence of one per 200 000 live
births [4–6]. The malformation is caused by a lack of regres-
sion of the cloacal membrane during the fourth week of
gestation [4,6,7]. This failure of mesenchymal cell migration
leads to an incomplete development of mesenchyme-derived
muscles and connective tissue of the abdominal anterior wall
of the bladder, with subsequent rupture of the anterior wall of
the bladder and exposure of the mucosa to the external
environment. These malformations may be associated with a
spectrum of other congenital defects, including skeletal pelvis
malformation and diastasis of the pubic symphysis. Urine’s
contact with the abdominal wall causes irritation and skin
maceration, whereas the bladder mucosa develops an
inflammatory reaction resulting in squamous metaplasia [7].
Early treatment of bladder exstrophy is focused on avoiding
other diseases of the urinary tract (like vesicoureteral reflux
with hydronephrosis, kidney infections, and bladder mucosa’s
cancer), ensuring bladder continence, and restoring the
external genitalia normal anatomy, avoiding personal hygiene
problems and severe psychosocial involvements for these
patients. Pelvic osteotomy was introduced, a little more than
50 years ago, into bladder exstrophy treatment protocols, and
it has led to a significant improvement in clinical outcomes
1060-152X Copyright © 2019 Wolters Kluwer Health, Inc. All rights reserved.
Copyright r 2019 Wolters Kluwer Health, Inc. All rights reserved.
Journal of Pediatric Orthopaedics B 2019, 28:207–213
Keywords: bladder exstrophy, congenital pelvic malformations,
oblique pelvic osteotomy
Departments of aOrthopaedics and Traumatology, bMedical and Surgical
Neonatology, cUrology and Robotic Surgery, Bambino Gesù Children’s Hospital,
Institute of Scientific Research, Rome and dUniversity of Cassino and Southern
Lazio, Cassino, Italy
Correspondence to Marco Giordano, MD, Department of Orthopaedics and
Traumatology, Bambino Gesù Children’s Hospital, Institute of Scientific Research,
Piazza S Onofrio, 4 Rome 00165, Italy
Tel: + 39 335 536 1549; fax: + 39 066 859 2195;
e-mail: marco.giordano@opbg.net
compared with those obtained with urological treatment
alone. The aim of orthopedic intervention is to close the
pelvic ring. It has functional effects on the urogenital tract and
facilitates urological reconstruction [2], eliminating the risk of
wound dehiscence and recurrent bladder prolapse. According
to a recent review [8], osteotomy, performed using various
techniques, was successful in ∼ 95% of patients (257/272) with
bladder exstrophy, with total absence of urological complica-
tions. Various authors [1,5,8–10] emphasize that there is a
direct relationship between execution of pelvic osteotomy,
whatever its mode, and the acquisition of bladder continence.
The aim of this study is to retrospectively analyze all cases of
newborns and infants affected by bladder exstrophy and
treated with oblique osteotomy, demonstrating that oblique
osteotomy analyzed in our series entailed a low risk of vascular
lesions or nerve damage, and it is a valid and safe alternative to
classic osteotomy.
Patients and methods
From November 2005 to January 2017, 20 patients with
bladder exstrophy were treated at our hospital. Two patients
were not included in this study because their follow-up timing
was too short (10 and 9 months). The remaining 18 neonatal
patients comprised 78% boys. The mean follow-up was
75 months (134–34 months). The patients’ parents gave us
written informed consent before the surgical procedure and
agreed for the infants to be included in this study. The
patients’ age at the time of the surgery ranged from 2 days to
3.5 months, with a mean age of 45 days (Table 1). An early
treatment of bladder exstrophy is indicated during the first
days of life, if the patient’s general condition allows it. In nine
cases (100% boys), surgery was performed electively a few
DOI: 10.1097/BPB.0000000000000614
208 Journal of Pediatric Orthopaedics B 2019, Vol 28 No 3

Table 1 Baseline characteristics of included patients in the study

PD
Urological Wound preoperative PD postoperative (mm) Loss of PD
Patients Sex Age at surgery procedure Follow-up T-T dehiscence UCa (mm) (follow-up 2 years) correction (%)

1 Male 3 days CPRE 11 years + 2 months No No Yes 29.85 10.81 36.2

2 Male 3 days CPRE 10 years + 10 months No No No 34.95 21.70 62
3 Male 5 days CPRE 9 years + 3 months No No Yes 41.84 22.72 54.3
4 Male 2 months DCPRE 8 years + 10 months Yes No Yes 37.03 20.50 55.3
5 Male 2 months + 1 day DCPRE 8 years + 2 months Yes No Yes 39.91 31.40 78.6
6 Male 1 month + 7 days DCPRE 7 years + 7 months Yes No Yes 39.94 23.15 57.9
7 Male 1 month + 28 days DCPRE 6 years + 9 months Yes No Yes 37.24 25.78 69.2
8 Male 3 months + 12 days DCPRE 6 years + 4 months Yes No Yes 31.15 19.20 61.6
9 Male 3 months + 17 days DCPRE 6 years + 2 months Yes No Yes 35.72 32 89.5
10 Female 8 days CPRE 5 years + 10 months No No Yes 31.20 22.80 73
11 Female 1 month + 17 days CPRE 5 years + 3 months No No Yes 36.50 24.08 65.9
12 Male 2 months + 4 days DCPRE 4 years + 10 months No No Yes 33.44 22.70 67.8
13 Female 10 days CPRE 4 years + 5 months No No Yes 27.94 16.31 58.3
14 Male 1 month + 6 days DCPRE 4 years + 3 months Yes No Yes 32.04 18.73 58.4
15 Female 14 days CPRE 4 years No No Yes 37.65 28.62 76
16 Female 1 month CPRE 3 years + 7 months No No Yes 34.50 21.51 62.3
17 Male 1 month + 5 days DCPRE 3 years Yes No Yes 36.91 32.4 87.7
18 Male 2 months + 23 days CPRE 2 years + 10 months Yes No NE 42.85 29.6 69

CPRE, complete primary repair of exstrophy; DCPRE, delayed complete primary repair of exstrophy; NE, not evaluable; PD, pubic diastasis (mm and percentage);
T-T, testosterone therapy; UC, urinary continence.
a
Dry time > 2 h.

weeks after birth, to permit presurgical pharmacological Fig. 1

treatment with testosterone of penis and glans epispadias.
Four intramuscular injections of testosterone enanthate
(100 mg/m2 of body surface area at 2-week intervals) and with
the child entrusted to the care of his/her parents at home, who
provided specific local treatment for the bladder mucosa, were
initiated. The aim of this treatment was for the patient to
reach an adequate body weight (4.5 kg, based on the
experience of neonatologists, anesthesiologists, and sur-
geons) so they can be in the best condition to undergo
surgery. In addition, hormonal treatment increases penile
volume and length, which helps the urological surgeon to
identify and manipulate the neurovascular bundles, pre-
venting possible damage that can result in tissue necrosis
[11,12]. The formerly mentioned three male patients were
younger than 1 week of life at surgery and did not received Three-dimensional computed tomography scan images of a newborn
pelvis on frontal view showing the direction for oblique pelvic osteotomy.
the testosterone treatment, because we still did not acquire
an adequate knowledge about it. Moreover, three female
infants were less than 2 weeks old at surgery. Three
patients underwent surgery ∼ 5 weeks of life, and other Fig. 2
three patients at ∼ 8 weeks after birth. One patient was
11 weeks old at surgery, and two patients finally were
treated at 14 and 15 weeks of life, correspondingly.

Surgical technique
The preoperative planning of the osteotomy was performed
using radiographs and computed tomography (CT) scan.
In three-dimensional view (Fig. 1), we could clearly observed
the relationship between the pelvis and internal organs.
Osteotomy was simulated on CT views to determine the level
and the inclination of the osteotomy. Under general anes-
thesia, the patients were placed in supine position (Fig. 2)
with a rolled towel inclined by ∼ 30° under the back to per- To perform the anterolateral approach, the patient is placed in supine
form the anterolateral approach. To facilitate the exposition of position. Before incision, a rolled towel was used to incline pelvis ∼ 30°.
the iliac wing, the surgical procedure began with a longitudinal

Copyright r 2019 Wolters Kluwer Health, Inc. All rights reserved.


skin incision of ∼ 4 cm practiced on the iliac crest, just prox-
imal to the anterior superior iliac spine (Fig. 3a). After peri-
osteum removal from the inner and outer surfaces of the ileum
and placing a spacer to protect the superior gluteal vessels and
nerve (Fig. 3b), an oblique section was performed using an
osteotome, from the top to the bottom of the iliac wing and
from the posterior to the acetabulum, extending to the greater
sciatic notch (Fig. 4a and b). The oblique pelvic osteotomy,
with internal and downward repositioning of the two front
sections of the pelvis and partial reconstruction of the pelvic
ring, allowed the urological surgeon to directly treat the
exstrophy, placing the bladder and urethra in the physiological
posterior position within the pelvic cavity. The urological
treatment was carried out respecting the criteria of complete
primary repair according to Grady and Mitchell [13] with
reconstruction of the bladder, the bladder neck, and the
genital organs, with restoration of the urethral retropubic
angle. Finally, the pubic diastasis was approached using
absorbable trans-osseous points, obtaining a pubic diastasis
width close to the normal value of 7 mm [14,15]. All surgeries
were performed by an experienced pediatric orthopedic
Fig. 3
(a) Intraoperative images. The skin incision is performed just proximal to the anterior superior iliac spine. (b) After removal of the periosteum, a spacer
is positioned in the outside of the iliac wing to protect vessels and nerve.
Fig. 4
Intraoperative images. (a, b) The oblique osteotomy is performed from the iliac crest to the greater sciatic notch.
Copyright r 2019 Wolters Kluwer Health, Inc. All rights reserved.
Pelvic osteotomy in bladder exstrophy Giordano et al. 209
surgeon and by the same team of urological surgeon and
neonatal surgeon.
Postoperative care
To maintain pelvic ring correction and to promote healing of
the osteotomy, patients wore a pelvic-podalic bilateral cast for
1 month postoperatively. The cast maintained the lower limbs
adducted and internally rotated and was equipped with a
removable half-pelvic ring in the front to allow urological
wounds and drainage examinations (Fig. 5). Postoperative
pain control was realized by a dedicated pediatric pain service.
Outcome examination
All patients were retrospectively evaluated clinically and
radiographically (Table 1). The follow-ups were performed at
regular intervals: 1, 2, 3, and 6 months after surgery, every 6
months until 2 years after surgery, and then every 12 months.
Physical examination and plain radiographs were obtained at
each visit to assess the outcome including bone healing,
nonunion, and infections. The functional outcome was
evaluated by an orthopedic surgeon according to the
patient’s age. Parameters evaluated were pain (according to
210 Journal of Pediatric Orthopaedics B 2019, Vol 28 No 3
parent’s surveys); functions as walking ability, gait and hip
range of movement (according to the gait and range of
movement sections of the Iowa Hip Score [16]; emotional
acceptance; and supports. Urological function was measured
both clinically (daytime dry intervals of 2 h or more) and
urodynamically (coordinated detrusor contractions). In
younger patients, urological function was measured by
voiding cystourethrogram on discharge after complete pri-
mary repair and repeated bladder ultrasound with report of
progressive increase of bladder capacity.
Statistical analysis
The Student t-test was used to compare pubic diastasis
before and after surgery. Data were analyzed with the
Fig. 5
Pelvipodalic plaster with soft anterior pelvic half-ring that allows
urological care. The lower limbs are adducted and internally rotated.
Fig. 6
Male newborn, 2 days old. (a) Preoperative pubic diastasis was 4.2 cm. (b) 6-year 8-month follow-up. A bilateral ‘step deformity’ of the iliac wings is
present (white arrows). The pubic diastasis is 2.1 cm, with normal relationship between femoral head and acetabulum.
Copyright r 2019 Wolters Kluwer Health, Inc. All rights reserved.
GraphPad Prisma Statistic Software (GraphPad Software,
San Diego, California, USA), and a cutoff of P value less
than 0.05 was considered significant.
Results and discussion
At follow-up examinations, there was an absence of pelvic
asymmetries, delayed or failed unions, or tenderness at the
sites of the intervention. All patients had no delay in starting
to walk (12 ± 3 months), reaching 10 points in the Iowa Hip
Score Gait section [16]. Orthopedic assessment showed
perfect wound healing. There were no signs of prior or
current bone infection. The cast was well tolerated, without
decubitus skin lesions. There was no recurrence of the
exstrophy with dehiscence of the abdominal wall. No leg
length discrepancy and lower limb axis deviation were
observed. All patients showed a flexion–extension range of
motion around 135° ± 5°, an abduction-adduction range of
80° ± 5°, and an internal–external rotation interval of 80° ± 5°.
Subjectively, no patient complained of pain at the osteotomy
site, either spontaneously or after pressure or palpation of the
iliac wings. Radiographically, a normal consolidation of the
osteotomies was observed in 100% of cases, with no signs of
skeletal dysplasia. It must be emphasized that reconstituting
the pelvic ring by rotating the anterior region of the pelvis
internally and down after the osteotomy entails a bilateral
alteration of the iliac wing profile that is characterized by an
apparent ‘bone step’ (Fig. 6). This anatomical and radio-
graphic modification has not been determined, after clinical
evaluation, to cause either aesthetic or functional problems.
The preoperative median pubic diastasis was 35.6 mm
(range: 27.94–42.85 mm, mean value 35.59 ± 0.9739). In
all patients, postoperative pubic diastasis was close to
7 mm. In all cases, there was a partial recurrence of dia-
stasis of the pubic symphysis during the first 6 months
after surgery. After that period, pubic diastasis became
stable.
The median pubic diastasis of these patients at 2-year
follow-up was 23.6 mm (range: 10.81–32.40 mm, mean
value 23.56 ± 1.347). The mean value of loss of correction

Fig. 7
Difference in pubic diastasis before and after surgery. Calculated means
and SD are shown for both groups.
was 65.7% (range: 89.5–36.2) 2 years after the surgery.
Although these data could indicate a failure, it is funda-
mental to focus on, for example, that a diastasis of 4 cm in
a newborn is more important compared with 4 cm at the
2-year follow-up because of the relative difference
between the diastasis and the diameter of the pelvis. The
difference between the two groups (before and after
surgery) was statistically significant (P < 0.0001, 95%
confidence interval: 8.657–15.42, R2 = 0.6067) (Fig. 7).
The loss of correction occurred because of the temporary
closure of the symphysis pubis owing to absorbable trans-
osseous suture. However, the temporary pelvic ring clo-
sure allowed stable replacement of the bladder–urethral
complex in a physiological position. The partial relapse of
pubic diastasis was not associated with recurrence of
bladder exstrophy or with clinically detectable functional
implications for the musculoskeletal system. A partial
relapse of pubic diastasis with growth has also been
reported by other authors [7,8,17]. Such relapse has not
been reported to adversely affect either the functional
outcome of the urinary tract or the motor activity of the
pelvis and lower limbs. Diastasis recurrence is less fre-
quent in older patients [18], which may be owing to the
lower growth of the ischiopubic segment compared with
the sacroiliac segment [5]. Other authors believe that
distracting forces caused by the pelvic muscles and liga-
ments and the volume increase of the intrapelvic organs
may be determinant for continence after surgery [19].
In addition to the absence of bladder exstrophy recurrence,
17 of the 18 patients treated in the neonatal period achieved
urinary continence (dry interval of ≥ 2 h) at a mean follow-up
of 4 years. Evaluation of urological functionality showed an
overall improvement in patients treated with oblique
osteotomy of the pelvis compared with past patients who
did not undergo this orthopedic intervention.
The main purpose of the orthopedic intervention is to
facilitate the success of reconstructive urogenital surgery.
Copyright r 2019 Wolters Kluwer Health, Inc. All rights reserved.
Pelvic osteotomy in bladder exstrophy Giordano et al. 211
The introduction of pelvic osteotomy over 50 years ago has
brought several important benefits that have increased the
effectiveness of the reconstruction carried out by urologists:
(a) reduction of pubic diastasis allows to restore the bladder
and urethra normal anatomy [7,17,20]; (b) owing to the
organs repositioning in the pelvis, skin tension decreases
and the risk of wound dehiscence and exstrophy recurrence
is very low [9,21]; and (c) to achieve urinary continence, it
is fundamental to restore the physiological position
of the urinary apparatus. The proximity of the pelvic floor
muscles, particularly the elevator ani muscle [7,10], com-
bined with the partial recovery of the periurethral and
perirectal muscles tonus, effectively contributes to con-
tinence. Moreover, in females, recovery of the pelvic
muscular integrity decreases the risk of late uterine prolapse
[8,13]. (d) In males, bringing closer together the two pubic
rami, using osteotomy, allows the apposition of the two
corpora cavernosa, facilitating reconstructive surgery of the
penis and a more anatomical structure of the genital tract [6,
18,22]. It could also reduce the risk of ischemic damage to
the penis [11]. In individuals with bladder exstrophy com-
pared with normal pelvis, CT revealed an average external
rotation of 12° of the posterior pelvis, measured by the
evaluation of the sacroiliac angle, which is an index of the
greater external inclination of the sacroiliac joint. There is
also an average external rotation of 18° of the anterior part of
the pelvis (the ischiopubic rami), measured by the angle
between a line joining the triradiate cartilages and a line
drawn through the pubic ramus between the center of the
acetabulum and the more medial point of the pubis [4].
The overall external rotation is ∼ 30°. Moreover, the ante-
rior segment of each half of the pelvis is shorter than the
normal (∼30%), and the acetabular retroversion is ∼ − 13°
(in comparison the normal value is 0°). In CT three-
dimensional reconstructions of patients’ pelvis with blad-
der exstrophy [23,24], a 15° increase was observed in the
rotation of the lower pelvis. In a study of Jani et al. [25],
radiographic data from adult patients with bladder exstro-
phy who were not treated with osteotomy (n = 12) or with
loss of correction after osteotomy (n = 2) were used for a
biomechanical analysis. The combination of longer distance
between the center of the femoral head and the midline of
the body, longer distance between the triradiate cartilages
(31% on average), and shorter distance between the greater
trochanter and femoral epiphysis center reduced the lever
arm and greatly increased the force required by the hip
abductor to maintain static equilibrium. The result of these
biomechanical changes increased the load and stress on
the hip joints with possible early onset of arthritic changes.
To our knowledge, there are no studies in literature
with a long-term follow-up to corroborate this hypothesis.
However, if an association will be confirmed, pelvic
osteotomy, even with partial correction of the described
parameters, could have a role in preventing degenerative
changes of the hip joint [8]. Some authors have reported
that patients with bladder exstrophy do not manifest par-
ticular orthopedic problems [8,17,21]. Others described a
212 Journal of Pediatric Orthopaedics B 2019, Vol 28 No 3
characteristic waddling gait [18] with external rotation of the
feet of 20°–30° [4], and difficult in accelerating the pace and
increasing step cadence [17]. Posterior osteotomy was the
first intervention proposed and remained such for almost
30 years [26]. The increasing knowledge of the pelvic
skeletal alterations and observation of surgical outcomes
have resolved the following major problems of this method:
the need of overcorrection in internal rotation to obtain an
effective pelvic ring closure [4], leg length difference [27],
pelvic pain and occasional delayed union after surgery [19],
significant blood loss, and the need to reposition patients
during surgery [9]. In 1991, Sponseller et al. [28] reported
the first analysis of horizontal osteotomy of the iliac wing.
The same group of authors [29] subsequently combined the
horizontal approach with a posterior osteotomy, which gave
immediate access to the lateral sacroiliac joint. Sponseller
et al. [5] stated that the posterior vertical osteotomy must be
adopted in patients over 2 years of age. The percentage of
failures (abdominal wound dehiscence or bladder prolapse)
was 4%, similar to one reported with posterior osteotomy
[1,2]. In 1994, McKenna et al. [30] presented their experi-
ence performing the four different known osteotomies,
concluding that diagonal iliac osteotomy is preferable
because it allows a more symmetric reconstruction of the
pelvis with less risk of surrounding structure injuries. Our
group preferred the iliac oblique osteotomy because the
surgical technique is, in our opinion, the simplest procedure
to perform. It requires a modest dissection of tissues, which
allows less blood loss, a lower risk of vascular and/or neu-
rological lesions, and a lower risk of infectious complications
than all the other techniques. The incision at the apex of
the iliac crest does not require special muscle dissection
because the elevation of the periosteum from the internal
and external iliac walls simultaneously separates the iliop-
soas and gluteal muscle insertions, respectively. The
exposure is limited to the upper margin of the greater sciatic
notch. In conclusion, a larger cohort of patients would be
useful to expand our data and further validate our results,
but the pathology is, luckily, a very rare condition.
However, we emphasize that this technique entailed a low
risk of vascular lesions or nerve damage, and it is a valid and
safe alternative to classic osteotomy. In our opinion,
osteotomy should be performed in all cases of bladder
exstrophy to eliminate the possibility of recurrence and to
improve the anatomical results of surgical reconstruction.
Repositioning the ischiopubic rami and positioning the
bladder and urethra within the pelvic osteotomy, restoring
of a normal bladder and urethral angle, and a more phy-
siological disposition of the pelvic floor muscles (interior
and exterior shutter, elevator) around the bladder neck and
urethra, are all important prerequisites to achieve con-
tinence [6,7,21].
When possible, we suggest performing the osteotomy
together with bladder reconstruction during the first
week of life. In male patients, surgery can be postponed
for a few weeks to allow the patient to achieve an
Copyright r 2019 Wolters Kluwer Health, Inc. All rights reserved.
adequate dimension of the genital structures with hor-
monal treatment. The option of postponing the treat-
ment of bladder exstrophy with no permanent injury and
with excellent functional results has recently been
described by other authors [31]. In neonatal period, a
plaster cast is the most effective way of postsurgical
immobilization because it promotes good bone healing
and it is well tolerated. Whatever preferences and options
are chosen for therapy, bladder exstrophy requires a
multidisciplinary approach and should be treated in
specialized centers with adequate surgical experience.
Acknowledgements
Conflicts of interest
There are no conflicts of interest.
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